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Lynch syndrome genes
1Department of Medical Genetics, Biomedicum Helsinki, University of Helsinki, P.O. Box 63, Haartmaninkatu 8, 00014, Finland. Paivi.Peltomaki@Helsinki.Fi
Abstract:
Since the discovery of the major human genes with DNA mismatch repair (MMR) function in 1993-1995, mutations in four, MSH2, MLH1, MSH6, and PMS2, have been convincingly linked to susceptibility of hereditary nonpolyposis colorectal cancer (HNPCC)/Lynch syndrome. Among these, PMS2 mutations are associated with diverse clinical features, including those of the Turcot syndrome. Two additional MMR genes, MLH3 and PMS1, have also been proposed to play a role in Lynch syndrome predisposition, but the clinical significance of mutations in these genes is less clear. According to the database maintained by the International Collaborative Group on Hereditary Nonpolyposis Colorectal Cancer (ICG-HNPCC), current InSiGHT (International Society for Gastrointestinal Hereditary Tumors), approximately 500 different HNPCC-associated MMR gene mutations are known that primarily involve MLH1 (approximately 50%), MSH2 (approximately 40%), and MSH6 (approximately 10%). Examination of HNPCC/Lynch syndrome-associated MMR genes and their mutations has revealed several other important functions for their protein products beyond postreplicative mismatch repair as well as many alternative mechanisms of pathogenicity. Despite these advances, much is yet to be learned about the molecular basis of correlations between genetic changes and clinical features of the disease.
Insights
Mutations in DNA mismatch repair (MMR) genes like MLH1 and MSH2 are linked to Lynch syndrome. Further research is needed to understand the molecular basis of genetic changes and their clinical features.
Area of Science:
- Genetics
- Oncology
- Molecular Biology
Background:
- DNA mismatch repair (MMR) gene mutations discovered between 1993-1995.
- Four MMR genes (MSH2, MLH1, MSH6, PMS2) are linked to hereditary nonpolyposis colorectal cancer (HNPCC)/Lynch syndrome.
- PMS2 mutations are associated with Turcot syndrome; MLH3 and PMS1 roles are less clear.
Purpose of the Study:
- To review known mutations in MMR genes associated with HNPCC/Lynch syndrome.
- To explore the functions of MMR gene products beyond DNA repair.
- To investigate the molecular basis of genotype-phenotype correlations in HNPCC/Lynch syndrome.
Main Methods:
- Database review of MMR gene mutations.
- Analysis of known functions of MMR gene products.
- Literature review on genotype-phenotype correlations.
Main Results:
- Approximately 500 HNPCC-associated MMR gene mutations are known.
- MLH1 (50%), MSH2 (40%), and MSH6 (10%) are the most frequently mutated genes.
- MMR genes have functions beyond DNA repair, and diverse pathogenicity mechanisms exist.
Conclusions:
- Significant advances have been made in understanding MMR gene mutations in HNPCC/Lynch syndrome.
- Further research is required to elucidate the molecular mechanisms linking genetic alterations to clinical manifestations.
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